Proboscis lateralis with ipsilateral sinonasal and olfactory pathway aplasia

Vaid, Sanjay; Shah, Darshan; Rawat, Sudarshan; Shukla, Rahul

doi:10.1016/j.jpedsurg.2009.10.044

Cited by 5 publications

(5 citation statements)

References 9 publications

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“…If this is the case, the philtrum, if present, may be the result of compensatory growth of the globular process of the normal unaffected side. This theory is further supported by the fact that on the affected side, there is atresia of the anterior nares and there is an absence of cribriform plate and olfactory bulb as shown on the results of T2-weighted images conducted on a patient with lateral proboscis (Vaid et al, 2010).…”

Section: Pathogenesismentioning

confidence: 76%

“…The role of the nasal placode in the development of the lateral proboscis remains at the center of our attention. The pathology that leads to aberration of the medial and lateral promay lead to the findings of absent olfactory bulb and olfactory sulcus on the affected side (Vaid et al, 2010). Before corrective surgery, CT and magnetic resonance imaging (MRI) should be performed to examine the connection of the lesion with neighboring structures and to present the precise anatomy, as well as to identify any brain abnormalities (Belet et al, 2002).…”

Section: Discussionmentioning

confidence: 99%

“…Diagnosis is made by clinical examination, nasal endoscopy, and confirmation with computed tomography (CT) scanning (Abou-Elhamd, 2004). Vaid et al (2010) reported a 2-year-old child with proboscis lateral in the left medial canthus who was scheduled for CT. A CT scan showed left sinonasal aplasia, with agenesis of the left nasal vault, maxillary, and ethmoid sinus. The proboscis arises from an osseous opening in the superomedial side of the left orbit.…”

Section: Diagnosismentioning

confidence: 99%

“…The incidence of this condition was reported to be around 1:100.000 to 1:1.000.000 live births with a male preponderance of 3:1 (Sakamoto et al, 2010; Verma et al, 2011; Magadum et al, 2012). However, patient history revealed no known history of facial abnormalities in the family and that karyotyping revealed no divergence from the normal 46XX or 46 XY karyotype (Martin et al, 2013).…”

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

A Case Report on Lateral Proboscis: A Rare Congenital Anomaly

Putri

Aditra

Apriawan

et al. 2021

The Cleft Palate Craniofacial Journal

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Lateral proboscis is a rare congenital condition characterized by a cylindrical protuberance on the nasofrontal region accompanied by abnormal nasal development on the affected side. We aimed to describe the management of the lateral proboscis in staged repair. A 7-year-old girl came with a tube-like projection on the left medial canthal region and nasal agenesis on the ipsilateral side. She was diagnosed with lateral proboscis, left microphthalmia, lower eye lid coloboma, and asymmetry in the orbital region. The patient has undergone 3 major surgeries at our institution. The first surgery involved the deconstruction of the tube to form the left nasal body and nostril. The second operation involved trimming of the new nose form and the excision of the bony protrusion directly beneath the base of the pedicle through bifrontal craniotomy. The remaining bone defect was closed using a pericranial flap. The orbital floor was reconstructed using titanium mesh. The third operation involved nasal reconstruction using a costal cartilage graft to create a dorsal nasal and alar framework. The patient healed with no complications, had become less reserved and her grades improved significantly after the operation. Further appointments are being scheduled to evaluate growth distortion and the resulting facial asymmetry. Surgical correction will be planned thereafter to further reconstruct the facial features. Evaluation of patient is necessary to explore possible clinical outcomes and corresponding treatment options. Multidisciplinary management is highly recommended, involving plastic surgeons, neurosurgeons, ophthalmologists, pediatricians, and pediatric psychiatrists in order to improve patient's quality of life.

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Section: Pathogenesismentioning

confidence: 76%

Section: Discussionmentioning

confidence: 99%

Section: Diagnosismentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

A Case Report on Lateral Proboscis: A Rare Congenital Anomaly

Putri

Aditra

Apriawan

et al. 2021

The Cleft Palate Craniofacial Journal

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show abstract

“…This type of proboscis is absent from true cyclops (Srinivasan et al, ; Yamada, Uwabe, Fujii, & Shiota, ). Notably, although the presence of proboscis is usually associated with cyclopia, this type of structure can also appear in noncyclopic human embryos ( proboscis lateralis , Figure g; Belet, Belet, Tekat, & Küçüködük, ; Vaid, Shah, Rawat, & Shukla, ). The incomplete formation of one side of the nose in cases of proboscis lateralis supports the hypothesis that the proboscis, which is placed laterally to the eye in this condition, is related to the embryonic anlage of the nose (Figure g).…”

Section: Discussionmentioning

confidence: 99%

Cellular identities in an unusual presentation of cyclopia in a chick embryo

2019

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Cyclopia is a congenital anomaly characterized by the presence of a single or partially divided eye in a single orbit at the body midline. This condition is usually associated with other severe facial malformations, such as the absence of the nose and, on rare occasions, the presence of a proboscis located above the ocular structures. The developmental origin of cyclopia in vertebrates is the failure of the embryonic prosencephalon to divide properly during the formation of the two bilateral eyes. Although the developmental origin of the cyclopia‐associated proboscis is not clear, it has been suggested that this unique structure results from the disrupted morphogenesis of the olfactory placodes, the main organizers of the developing nose. In this study, we report a spontaneous congenital case of cyclopia with a proboscis‐like appendage in a chick embryo. By means of both conventional histology and immunohistochemical methods, we have analyzed this anomaly in detail to suggest an alternative identity for the anatomical embryonic features of cyclopic vertebrate embryos displaying a proboscis. Our findings are discussed in the context of previously reported cases of cyclopia, and provide additional insight into this complex congenital malformation.

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Anatomy of Mesenchyme and the Pharyngeal Arches

Carstens

2023

The Embryologic Basis of Craniofacial Structure

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Proboscis lateralis with ipsilateral sinonasal and olfactory pathway aplasia

Cited by 5 publications

References 9 publications

A Case Report on Lateral Proboscis: A Rare Congenital Anomaly

A Case Report on Lateral Proboscis: A Rare Congenital Anomaly

Cellular identities in an unusual presentation of cyclopia in a chick embryo

Anatomy of Mesenchyme and the Pharyngeal Arches

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